Disseminated Xanthogranuloma Harboring a GAB2::BRAF Fusion and MITF Pathogenic Variant Successfully Treated With Cobimetinib: A Rare Case Report.

IF 1.2 4区 医学 Q3 DERMATOLOGY
Pauline C Xu, Allison M Cushman-Vokoun, Christopher R D'Angelo, Dinesh Pradhan
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Abstract

Several activating mutations and gene fusions involving the mitogen-activated protein kinase (MAPK) pathway have appeared in the literature regarding histiocytic neoplasms. We identified a GAB2::BRAF fusion in a cutaneous lesion of a 36-year-old male who developed central diabetes insipidus and reddish-pink grouped papules on the bilateral axillary rims, inguinal region, and periocular skin. Skin biopsy obtained from an axillary lesion showed a CD68+, S100-, and CD1a- histiocytic proliferation with foamy histiocytes and multinucleated Touton giant cells, compatible with xanthogranuloma. Next-generation sequencing identified a GAB2::BRAF fusion involving Exon 2 of GAB2 and Exon 10 of BRAF. Our case further highlights this novel fusion in the MAPK signaling pathway as a possible driver of nonLangerhans cell histiocytosis (NLCH) and underscores the utility of performing molecular studies on skin biopsy specimens with NLCH to help identify potential targets for therapy.

Cobimetinib成功治疗含有GAB2::BRAF融合和MITF致病变异的弥散性黄色肉芽肿:一例罕见病例报告。
一些涉及丝裂原活化蛋白激酶(MAPK)途径的激活突变和基因融合已经出现在有关组织细胞肿瘤的文献中。我们在一名36岁男性患者的皮肤病变中发现了GAB2::BRAF融合,该患者发展为中枢性尿囊症,并在双侧腋窝边缘、腹股沟区域和眼周皮肤上发现了红粉色组丘疹。腋窝病变的皮肤活检显示CD68+、S100-和CD1a-组织细胞增生,伴有泡沫组织细胞和多核图顿巨细胞,与黄色肉芽肿相容。下一代测序发现GAB2::BRAF融合涉及GAB2的外显子2和BRAF的外显子10。我们的病例进一步强调了MAPK信号通路中的这种新型融合作为非朗格汉斯细胞组织细胞增生症(NLCH)的可能驱动因素,并强调了对具有NLCH的皮肤活检标本进行分子研究以帮助确定潜在治疗靶点的实用性。
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来源期刊
CiteScore
3.20
自引率
5.90%
发文量
174
审稿时长
3-8 weeks
期刊介绍: Journal of Cutaneous Pathology publishes manuscripts broadly relevant to diseases of the skin and mucosae, with the aims of advancing scientific knowledge regarding dermatopathology and enhancing the communication between clinical practitioners and research scientists. Original scientific manuscripts on diagnostic and experimental cutaneous pathology are especially desirable. Timely, pertinent review articles also will be given high priority. Manuscripts based on light, fluorescence, and electron microscopy, histochemistry, immunology, molecular biology, and genetics, as well as allied sciences, are all welcome, provided their principal focus is on cutaneous pathology. Publication time will be kept as short as possible, ensuring that articles will be quickly available to all interested in this speciality.
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